CASE REPORTS so far described and not in pediatric patients as per Funding: None. Competing interest: None stated. available English literature. REFERENCES

A large number of adverse reactions to IVIG have been 1. Rizk A, Gorson KC, Kenney L, Weinstein R. Transfusion- reported [4-7], including hypersensitivity reaction like related acute lung injury after the infusion of IVIG. urticaria, angioedema and bronchospasm. Respiratory Transfusion. 2001;41:264-8. distress following any infusion can also occur because of 2. Kleinman S, Caulfield T, Chan P, Davenport R, McFarland circulatory overload or anaphylactic transfusion reactions. J, McPhedran S, et al. Toward an understanding of Transfusion-associated circulatory overload develops transfusion-related acute lung injury: statement of a within minutes to hours of transfusion as congestive consensus panel. Transfusion. 2004;44:1774-89. cardiac failure, cyanosis, hypertension and it rapidly 3. Suassuna JHR, da Costa MADL, Faria RA, Melichar AC. Noncardiogenic pulmonary edema triggered by responds to aggressive diuresis and ventilatory support. intravenous immunoglobulin in cancer – associated Anaphylactic transfusion reactions results in laryngeal and thrombotic thrombocytopenic purpura- hemolytic uremic bronchial spasm and manifest as tachypnea, wheezing, syndrome. Nephron. 1997;77:368-70. cyanosis, erythma, edema and severe hypotension, during 4. Orbach H, Katz U, Sherer Y, Shoenfeld Y. Intravenous the transfusion of any type of protein-containing blood immunoglobulin: adverse effects and safe administration. component [8]. Clin Rev Allergy Immunol. 2005;29:173-84. 5. Berkovitch M, Dolinski G, Tauber T, Aladjem M, Akin to post blood component infusion TRALI, that Kaplinsky C. Neutropenia as a complication of intravenous following IVIG infusion could have been due to immune immunoglobulin (IVIG) therapy in children with immune mediated mechanism [9] or priming thrombocytopenic purpura: common and non-alarming. Int mechanism [2]. In our patient, the possible mechanism J Immunopharmacol. 1999;21:411-15. 6. Daphnis E, Stylianou K, Alexandrakis M, Xylouri I, could not be ascertained since the estimation of antibody Vardaki E, Stratigis S, et al. Acute renal failure, against was not available. translocational hyponatremia and hyperkalemia following intravenous immunoglobulin therapy. Nephron Clin Pract. It is suggested that clinician using IVIG should closely 2007;106:c143-8. monitor the patients to pick up this potential fatal 7. Shorr AF, Kester KE. Meningitis and hepatitis complication at the earliest and institute appropriate complicating intravenous immunoglobulin therapy. Ann supportive care timely. Pharmacother. 1996;30:1115-6. 8. Silliman CC, Ambruso DR and Boshkov LK. Transfusion Contributors: VG and TPY were involved in active case – related acute lung injury. Blood. 2005:105:2266-73. management and review of literature. VG and PG revised the 9. Moalic V, Vaillant C, Ferec C . Transfusion related acute paper for critically important intellectual content. All authors lung injury (TRALI): an unrecognised pathology. Pathol approved the final version to be published. Biol. 2005;53:111-5.

Kawasaki Disease in Association with Urinary Tract

ENTESAR H HUSAIN†‡ AND #MARYAM A L-RASHID

From the †Department of Pediatrics, Faculty of Medicine; ‡Department of Pediatrics, Mubarak Al-Kabeer Hospital; #Department of Pediatrics, Amiri Hospital; Kuwait.

Correspondence to: We report a 2-month-old infant with E. coli , who did not Dr Entesar H Husain, respond to therapy. She later developed clinical features fulfilling criteria Department of Pediatrics, Faculty of of (KD), and was treated with intravenous immunolglobulin and Medicine, P.O. Box 24923, Safat, aspirin. KD should be considered in the differential diagnosis in patients who Kuwait 13110. [email protected] present with infection and do not respond to antibiotic therapy. Received: March 03, 2010; Initial review: April 06, 2010; Key words: Kawasaki disease, Urinary tract infection. Accepted: July 26, 2010.

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awasaki disease (KD) is an acute febrile abnormal urinary findings such as sterile pyuria, mild vasculitis of childhood characterized by the and microscopic hematuria. Sterile pyuria has following clinical features; bilateral non- been reported to occur in 10-50% of children during the Kexudative conjunctivitis, erythema of the lips acute phase [1]. It was well known that pyuria in KD and oral mucosa, changes in the extremities, rash and originate from the urethra [3], but a recent study showed cervical lymphadenopathy. Sterile pyuria has been seen in that the white blood cells in the urine in patients with KD 10-50% of patients with KD in the acute phase [1]. might originate from the urethra or the kidney or both [4]. Recently, some reports have shown the association of KD In most of the described series, sterile pyuria is found more with urinary tract infection (UTI) [1,2]. commonly in infants than in older children [5]. We report an infant with KD with UTI in whom the In addition to the current case, there are two cases in clinical manifestations of KD evolved later. the literature of children diagnosed with a documented CASE REPORT UTI who subsequently had KD [1,2]. It is unclear if the vasculitis is provoking the infection or the gram negative A previously healthy 2 ½ -month old infant, presented with organisms precipitating UTI are able to produce a 2-day-history of a febrile illness associated with superantigens similar to staphylococcal and streptococcal maculopapular rash and nasal congestion. She also had a antigens that have been suggested to be responsible for the history of irritability and decreased feeding for one day development of KD. From the cases described, UTI seems prior to admission. On examination, she was unwell and to be more common in infants with incomplete features of irritable. Her temperature was 39.5ºC, heart rate 145/ KD. It is difficult to conclude from the limited number of minute and respiratory rate 35 per minute. The cases if infants with underlying vesicoureteric reflux are cardiovascular, respiratory and abdominal examination more likely to present with UTI when presenting with KD. was within normal. She had severe nasal block and tearing The likelihood of developing coronary aneurysms of the right eye but no conjunctivitis. A generalized increases in children who had UTI due to misdiagnosis and maculopapular rash was noted over the face, extremities the delay in administering IVIG [2]. and trunk. The Initial investigations showed hemoglobin 84 g/L, platelets 388×109 /L, and white blood cell count In conclusion, it is unclear if UTI is a cause of KD in 10.3×109 /L (60% neutrophils, 32% lymphocytes, 7% some infants or this observation is only coincidental. monocytes, 1% eosinophils). The C-reactive protein was Laboratory evidence of infection in a patient does not 57 mg/L and liver function tests were normal. Urinalysis always rule out KD. KD should be one of the differential showed WBC 6-8 cells /HPF and urine nitrite positive; diagnoses in patients who are suspected of having UTI and urine culture grew 105 CFU/mL of E.coli. She received do not respond to antibiotic therapy. treatment with intravenous cefotaxime (150 mg/kg/day). Contributors: Both authors were involved in all aspects of On the fourth day of admission, the child was still irritable, patient management and manuscript preparation. with decreased oral feeding and febrile at 39ºC; swelling of Funding: None. both hands and feet was noted with accentuated erythema. Competing interests: None stated. She had also developed cracked lips with bleeding and bilateral conjunctivitis. The baby was diagnosed to have REFERENCES KD based on the presence of the clinical features and was 1. Shiono N, Koga Y, Ito H, Egawa K, Ono S, Itami N. Really started on intravenous immunoglobulin 2 g/kg, and aspirin sterile pyuria with Kawasaki disease. Pediatr Nephrol. 100 mg/kg/day. The fever subsided after 48 hours and there 2004;19:124. was a marked improvement of the irritability and feeding 2. Wu CY, Hsieh KS, Chiou YH, Wang RS, Huang IF, Lee pattern. Cefotaxime was continued for treatment of the WY, et al. Prolonged fever and pyuria: a urinary tract UTI. Subsequently on the 9th day of the illness, she infection presentation of incomplete Kawasaki disease. developed peeling of the skin overlying the tips of her Acta Paediatr. 2005;3:375-7. fingers and toes. Echocardiography, done during 3. Melish ME, Hicks RM, Larson EJ. Mucocutaneous lymph hospitalization and follow up, was normal. Follow up blood node syndrome in the United States. Am J Dis Child. 1976;130:599-607. counts on the 10th day of admission showed increasing 4. Watanabe T, Abe Y, Sato S, Uehara Y, Ikeno K, Abe T. 9 platelets counts to 722×10 /L. An ultrasound scan revealed Sterile pyuria in patients with Kawasaki diseases originates normal kidneys and a voiding cyctourethrogram four from both the urethra and kidney. Pediatr Nephrol. weeks after discharge excluded vesicoureteric reflux. 2007;7:987-91. 5. Wirojanan J, Sopontammarak S, Vachvanichsanong P. DISCUSSION Sterile pyuria in Kawasaki disease. Pediatr Nephrol. KD is a systemic vasculitis, which may be associated with 2004;19:363.

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